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Validating the RedMIT/GFP-LC3 mouse model by studying mitophagy in autosomal dominant optic atrophy due to the OPA1Q285STOP mutation

Abstract:

Background

Autosomal dominant optic atrophy (ADOA) is usually caused by mutations in the essential gene, OPA1. This encodes a ubiquitous protein involved in mitochondrial dynamics, hence tissue specificity is not understood. Dysregulated mitophagy (mitochondria recycling) is implicated in ADOA, being increased in OPA1 patient fibroblasts. Furthermore, autophagy may be increased in retinal ganglion cells (RGCs) of the OPA1Q285STOP mouse model.

Aims

We developed a m...

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Publication status:
Published
Peer review status:
Peer reviewed
Version:
Publisher's version

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Publisher copy:
10.3389/fcell.2018.00103

Authors


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Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
Pathology Dunn School
Role:
Author
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Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
Women's and Reproductive Health
Role:
Author
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Funding agency for:
Daniels, MJ
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Funding agency for:
Dombi, E
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Publisher:
Frontiers Media Publisher's website
Journal:
Frontiers in Cell and Developmental Biology Journal website
Volume:
6
Issue:
SEP
Pages:
103
Publication date:
2018-09-19
Acceptance date:
2018-08-13
DOI:
EISSN:
2296-634X
Pubs id:
pubs:923640
URN:
uri:6c9e1121-02b2-42e2-a206-2ccd3ef3f0d1
UUID:
uuid:6c9e1121-02b2-42e2-a206-2ccd3ef3f0d1
Local pid:
pubs:923640

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